{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,8]],"date-time":"2026-10-08T22:17:12Z","timestamp":1791497832572,"version":"4.3.4"},"reference-count":24,"publisher":"MDPI AG","issue":"24","license":[{"start":{"date-parts":[[2023,12,13]],"date-time":"2023-12-13T00:00:00Z","timestamp":1702425600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["JCM"],"abstract":"<jats:p>Severe hemolytic disease of the fetus and newborn (HDFN) requiring intrauterine transfusions (IUTs) may cause iron accumulation, resulting in liver damage, which may lead to cholestasis and coagulation disorders. In this article, we reported a case of a female neonate who underwent chelation therapy with a positive outcome, and we reviewed the English and Polish literature on chelation therapy in HDFN available in PubMed. The patient with maximum ferritin concentration above 33,511.2 ng\/mL developed liver dysfunction with coagulation disorders requiring multiple transfusions of fresh frozen plasma (FFP), Octaplex\u00ae and cryoprecipitate, and hypoalbuminemia treated with numerous albumin infusions. Furthermore, severe cholestasis was observed with direct bilirubin levels up to 33.14 mg\/dL. Additionally, the child developed transient myelosuppression with neutropenia, thrombocytopenia, and low reticulocyte count due to several blood transfusions. The differential diagnosis tests were conducted to rule out any causes of hepatic failure other than hemolytic disease of the newborn. This case proves that adequate treatment of severe HDFN with anemia requiring IUT and hepatic failure can lead to positive outcomes with no long-term consequences.<\/jats:p>","DOI":"10.3390\/jcm12247645","type":"journal-article","created":{"date-parts":[[2023,12,13]],"date-time":"2023-12-13T08:55:16Z","timestamp":1702457716000},"page":"7645","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":6,"title":["Liver Dysfunction with Severe Cholestasis and Coagulation Disorders in the Course of Hemolytic Disease of the Newborn Requiring Chelation Therapy\u2014A Case Report and Review of the Literature"],"prefix":"10.3390","volume":"12","author":[{"ORCID":"https:\/\/orcid.org\/0009-0003-3503-6660","authenticated-orcid":false,"given":"Agnieszka","family":"Drozdowska-Szymczak","sequence":"first","affiliation":[{"name":"Department of Neonatology and Neonatal Intensive Care, Institute of Mother and Child, Kasprzaka 17a, 01-211 Warsaw, Poland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0000-4852-3298","authenticated-orcid":false,"given":"Julia","family":"Proczka","sequence":"additional","affiliation":[{"name":"Department of Neonatology and Neonatal Intensive Care, Institute of Mother and Child, Kasprzaka 17a, 01-211 Warsaw, Poland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Danuta","family":"Chrzanowska-Liszewska","sequence":"additional","affiliation":[{"name":"Department of Neonatology and Neonatal Intensive Care, Institute of Mother and Child, Kasprzaka 17a, 01-211 Warsaw, Poland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Krzysztof","family":"Truszkowski","sequence":"additional","affiliation":[{"name":"Department of Neonatology and Neonatal Intensive Care, Institute of Mother and Child, Kasprzaka 17a, 01-211 Warsaw, Poland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6970-5303","authenticated-orcid":false,"given":"Natalia","family":"Mazanowska","sequence":"additional","affiliation":[{"name":"Department of Obstetrics and Gynecology, Institute of Mother and Child, Kasprzaka 17a, 01-211 Warsaw, Poland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Pawe\u0142","family":"Krajewski","sequence":"additional","affiliation":[{"name":"Department of Neonatology and Neonatal Intensive Care, Institute of Mother and Child, Kasprzaka 17a, 01-211 Warsaw, Poland"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1968","published-online":{"date-parts":[[2023,12,13]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"99","DOI":"10.1111\/vox.12265","article-title":"Haemolytic disease of the fetus and newborn","volume":"109","author":"Thurik","year":"2015","journal-title":"Vox Sang."},{"key":"ref_2","doi-asserted-by":"crossref","first-page":"1099","DOI":"10.1002\/pd.5674","article-title":"Red cell alloimmunization: A 2020 update","volume":"40","author":"Castleman","year":"2020","journal-title":"Prenat Diagn."},{"key":"ref_3","doi-asserted-by":"crossref","first-page":"263","DOI":"10.1159\/000362812","article-title":"Intrauterine blood transfusion: Current indications and associated risks","volume":"36","author":"Lindenburg","year":"2014","journal-title":"Fetal Diagn. Ther."},{"key":"ref_4","doi-asserted-by":"crossref","first-page":"328","DOI":"10.1111\/vox.12061","article-title":"Iron status in infants with alloimmune haemolytic disease in the first three months of life","volume":"105","author":"Rath","year":"2013","journal-title":"Vox Sang."},{"key":"ref_5","doi-asserted-by":"crossref","first-page":"204","DOI":"10.5152\/tjh.2010.30","article-title":"Severe iron overload and hyporegenerative anemia in a case with rhesus hemolytic disease: Therapeutic approach to rare complications","volume":"27","author":"Arslan","year":"2010","journal-title":"Turk. J. Haematol."},{"key":"ref_6","doi-asserted-by":"crossref","first-page":"e20182859","DOI":"10.1542\/peds.2018-2859","article-title":"Severe Fetal Hemolysis and Cholestasis Due to High-Titer Maternal IgG Anti-A Antibodies","volume":"143","author":"Zonneveld","year":"2019","journal-title":"Pediatrics"},{"key":"ref_7","doi-asserted-by":"crossref","first-page":"306","DOI":"10.1159\/000335333","article-title":"Cholestasis in neonates with red cell alloimmune hemolytic disease: Incidence, risk factors and outcome","volume":"101","author":"Rath","year":"2012","journal-title":"Neonatology"},{"key":"ref_8","first-page":"90","article-title":"The use of chelation therapy in the treatment of iron overload in a girl with Diamond-Blackfan anemia\u2014A case report","volume":"2011","author":"Romiszewski","year":"2011","journal-title":"Borgis-Nowa Pediatr."},{"key":"ref_9","doi-asserted-by":"crossref","first-page":"141","DOI":"10.1159\/000343261","article-title":"Neonatal morbidity after exchange transfusion for red cell alloimmune hemolytic disease","volume":"103","author":"Rath","year":"2013","journal-title":"Neonatology"},{"key":"ref_10","doi-asserted-by":"crossref","first-page":"660","DOI":"10.1038\/jp.2012.71","article-title":"An approach to the management of hyperbilirubinemia in the preterm infant less than 35 weeks of gestation","volume":"32","author":"Maisels","year":"2012","journal-title":"J. Perinatol."},{"key":"ref_11","doi-asserted-by":"crossref","first-page":"607","DOI":"10.1080\/17474086.2017.1331124","article-title":"Neonatal management and outcome in alloimmune hemolytic disease","volume":"10","author":"Ree","year":"2017","journal-title":"Expert. Rev. Hematol."},{"key":"ref_12","doi-asserted-by":"crossref","first-page":"215","DOI":"10.1016\/j.clp.2016.01.001","article-title":"Hyperbilirubinemia in Preterm Neonates","volume":"43","author":"Bhutani","year":"2016","journal-title":"Clin. Perinatol."},{"key":"ref_13","doi-asserted-by":"crossref","first-page":"702","DOI":"10.1038\/s41372-022-01345-1","article-title":"Population-based incidence and risk factors for cholestasis in hemolytic disease of the fetus and newborn","volume":"42","author":"Teng","year":"2022","journal-title":"J. Perinatol."},{"key":"ref_14","doi-asserted-by":"crossref","first-page":"154","DOI":"10.1097\/MPG.0000000000001334","article-title":"Guideline for the Evaluation of Cholestatic Jaundice in Infants: Joint Recommendations of the North American Society for Pediatric Gastroenterology, Hepatology, and Nutrition and the European Society for Pediatric Gastroenterology, Hepatology, and Nutrition","volume":"64","author":"Fawaz","year":"2017","journal-title":"J. Pediatr. Gastroenterol. Nutr."},{"key":"ref_15","first-page":"7","article-title":"Recommendation of early diagnosis of cholestasis in infant","volume":"14","author":"Jankowska","year":"2017","journal-title":"Stand. Med.\/Pediatr."},{"key":"ref_16","doi-asserted-by":"crossref","first-page":"763","DOI":"10.1148\/radiol.2017170305","article-title":"Hepatic Imaging in Neonates and Young Infants: State of the Art","volume":"285","author":"Shamir","year":"2017","journal-title":"Radiology"},{"key":"ref_17","doi-asserted-by":"crossref","first-page":"e63","DOI":"10.1542\/neo.14-2-e63","article-title":"Neonatal Cholestasis","volume":"14","author":"Feldman","year":"2013","journal-title":"Neoreviews"},{"key":"ref_18","doi-asserted-by":"crossref","first-page":"73","DOI":"10.1097\/MPH.0000000000001586","article-title":"Conservative Management of Hyperferritinemia in Hemolytic Disease of the Fetus and Newborn: A Case Report and Review of the Literature","volume":"43","author":"Adam","year":"2021","journal-title":"J. Pediatr. Hematol. Oncol."},{"key":"ref_19","doi-asserted-by":"crossref","first-page":"335","DOI":"10.24953\/turkjped.2018.03.018","article-title":"Chelation therapy for secondary neonatal iron over load: Lessons learned from rhesus hemolytic disease","volume":"60","author":"Aladily","year":"2018","journal-title":"Turk. J. Pediatr."},{"key":"ref_20","doi-asserted-by":"crossref","first-page":"290","DOI":"10.1097\/01.mph.0000212906.07018.93","article-title":"A case of rhesus hemolytic disease with hemophagocytosis and severe iron overload due to multiple transfusions","volume":"28","author":"Yilmaz","year":"2006","journal-title":"J. Pediatr. Hematol. Oncol."},{"key":"ref_21","doi-asserted-by":"crossref","first-page":"509","DOI":"10.1038\/sj.jp.7200458","article-title":"Successful chelation therapy in a case of neonatal iron overload following intravascular intrauterine transfusion","volume":"20","author":"Sreenan","year":"2000","journal-title":"J. Perinatol."},{"key":"ref_22","doi-asserted-by":"crossref","first-page":"1393","DOI":"10.1016\/j.pcl.2013.09.003","article-title":"Evaluation and treatment of transfusional iron overload in children","volume":"60","author":"Ware","year":"2013","journal-title":"Pediatr. Clin. N. Am."},{"key":"ref_23","doi-asserted-by":"crossref","first-page":"1457","DOI":"10.1007\/s00431-011-1521-7","article-title":"Desferrioxamine treatment of iron overload secondary to RH isoimmunization and intrauterine transfusion in a newborn infant","volume":"170","author":"Yalaz","year":"2011","journal-title":"Eur. J. Pediatr."},{"key":"ref_24","first-page":"41","article-title":"Management of cholestasis in infants","volume":"1","author":"Jankowska","year":"2013","journal-title":"Stand. Med.\/Pediatr."}],"container-title":["Journal of Clinical Medicine"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.mdpi.com\/2077-0383\/12\/24\/7645\/pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,10,10]],"date-time":"2025-10-10T21:37:40Z","timestamp":1760132260000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.mdpi.com\/2077-0383\/12\/24\/7645"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2023,12,13]]},"references-count":24,"journal-issue":{"issue":"24","published-online":{"date-parts":[[2023,12]]}},"alternative-id":["jcm12247645"],"URL":"https:\/\/doi.org\/10.3390\/jcm12247645","relation":{},"ISSN":["2077-0383"],"issn-type":[{"value":"2077-0383","type":"electronic"}],"subject":[],"published":{"date-parts":[[2023,12,13]]}}}